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Bang OY, Fujimura M. Medical Management of Adult Moyamoya Disease: A Review and Relevant Cases With Ischemic Events. J Stroke 2025; 27:1-18. [PMID: 39916450 PMCID: PMC11834343 DOI: 10.5853/jos.2024.04273] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/16/2024] [Revised: 11/01/2024] [Accepted: 11/20/2024] [Indexed: 02/20/2025] Open
Abstract
Moyamoya disease (MMD) is a rare and progressive cerebrovascular disorder characterized by stenosis or occlusion of the internal carotid arteries resulting in the development of fragile collateral vessels at the base of the brain. Surgical revascularization is the primary treatment option for preventing ischemic and hemorrhagic events; however, the role of medical management has become increasingly recognized, particularly in cases involving asymptomatic patients or those at a high risk for surgical complications. In this review, we aimed to investigate the current guidelines and evidence supporting various medical management strategies for MMD, including the importance of controlling risk factors and judicious use of antithrombotic therapy. Given the considerable variability in patient presentation, such as age of onset, symptomatology, and comorbid conditions, it is crucial to adopt tailored therapeutic approaches that address each patient's unique characteristics. The existing literature on medical management is limited. However, individualized strategies may effectively mitigate the risk of ischemic events and improve the overall patient outcomes. Further research is essential to develop comprehensive and standardized treatment protocols for medical management of adult patients with MMD. In addition, ongoing trials and efforts to develop disease-modifying agents are discussed.
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Affiliation(s)
- Oh Young Bang
- Department of Neurology, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Korea
- S&E bio Co., Ltd., Seoul, Korea
| | - Miki Fujimura
- Department of Neurosurgery, Hokkaido University Graduate School of Medicine, Sapporo, Japan
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Zhang G, Liu E, Tan X, Liu C, Yang S. Research progress on moyamoya disease combined with thyroid diseases. Front Endocrinol (Lausanne) 2023; 14:1233567. [PMID: 37900127 PMCID: PMC10600369 DOI: 10.3389/fendo.2023.1233567] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/05/2023] [Accepted: 09/04/2023] [Indexed: 10/31/2023] Open
Abstract
Moyamoya disease (MMD), also known as abnormal cerebral vascular network disease, is characterized by progressive occlusion or stenosis of the internal carotid and cerebral arteries, as well as the formation of an abnormal cerebral vascular network. It can occur anywhere in the world but is most common in China, Japan, and the Republic of Korea. In recent years, there have been increasing reports on the coexistence of thyroid diseases and MMD, but the mechanism of their coexistence is still unclear. For this article, we used keywords such as "moyamoya disease", "thyroid", "Grave disease", "thyrotoxicosis", and "thyroid autoimmune antibodies" to search for 52 articles that met the requirements in medical databases such as PubMed and Web of Science. This article also reviews the research on the role of thyroid hormone, the mechanism of immune antibodies, the possible correlation between thyroid diseases and MMD disease genes, and the treatment methods, and discusses the possible relationship between MMD and thyroid diseases to provide a reference for the pathogenesis and treatment of MMD with thyroid diseases.
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Affiliation(s)
- Guibo Zhang
- The Affiliated Hospital of Kunming University of Science and Technology, Kunming, China
- The First People’s Hospital of Yunnan Province, Kunming, China
| | - Erheng Liu
- Department of Neurosurgery, Kaiyuan Municipal People’s Hospital, Kaiyuan, China
| | - XueYi Tan
- The Affiliated Hospital of Kunming University of Science and Technology, Kunming, China
- The First People’s Hospital of Yunnan Province, Kunming, China
| | - Chengyuan Liu
- The Affiliated Hospital of Kunming University of Science and Technology, Kunming, China
- The First People’s Hospital of Yunnan Province, Kunming, China
| | - Shuaifeng Yang
- The Affiliated Hospital of Kunming University of Science and Technology, Kunming, China
- The First People’s Hospital of Yunnan Province, Kunming, China
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Skandalakis GP, Kalyvas A, Lani E, Komaitis S, Manolakou D, Chatzopoulou D, Pantazis N, Zenonos GA, Hadjipanayis CG, Stranjalis G, Koutsarnakis C. Effectiveness of pharmacologic interventions for prevention of cerebral hyperperfusion syndrome following bypass surgery. Brain Circ 2022; 8:207-214. [PMID: 37181839 PMCID: PMC10167845 DOI: 10.4103/bc.bc_43_22] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/07/2022] [Revised: 10/25/2022] [Accepted: 10/28/2022] [Indexed: 12/14/2022] Open
Abstract
BACKGROUND Cerebral hyperperfusion syndrome (CHS) following bypass surgery is a major cause of neurological morbidity and mortality. However, data regarding its prevention have not been assorted until date. OBJECTIVE The objective of this study was to review the literature and evaluate whether any conclusion can be drawn regarding the effectiveness of any measure on preventing bypass-related CHS. METHODS We systematically reviewed PubMed and Cochrane Library from September 2008 to September 2018 to collect data regarding the effectiveness of pharmacologic interventions on the refers to pretreatment (PRE) of bypass-related CHS. We categorized interventions regarding their class of drugs and their combinations and calculated overall pooled estimates of proportions of CHS development through random-effects meta-analysis of proportions. RESULTS Our search yielded 649 studies, of which 23 fulfilled inclusion criteria. Meta-analysis included 23 studies/2,041 cases. In Group A (blood pressure [BP] control), 202 out of 1,174 pretreated cases developed CHS (23.3% pooled estimate; 95% confidence interval [CI]: 9.9-39.4), Group B (BP control + free radical scavenger [FRS]) 10/263 (0.3%; 95% CI: 0.0-14.1), Group C (BP control + antiplatelet) 22/204 (10.3%; 95% CI: 5.1-16.7), and Group D (BP control + postoperative sedation) 29/400 (6.8%; 95% CI: 4.4-9.6)]. CONCLUSIONS BP control alone has not been proven effective in preventing CHS. However, BP control along with either a FRS or an antiplatelet agent or postoperative sedation seems to reduce the incidence of CHS.
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Affiliation(s)
- Georgios P. Skandalakis
- Athens Microneurosurgery Laboratory, Department of Neurosurgery, University of Athens, Athens, Greece
- Department of Neurosurgery, Icahn School of Medicine, Mount Sinai Health System, New York, NY, USA
| | - Aristotelis Kalyvas
- Athens Microneurosurgery Laboratory, Department of Neurosurgery, University of Athens, Athens, Greece
- Department of Neurosurgery, Toronto Western Hospital, University Health Network, Toronto, Ontario, Canada
| | - Evgenia Lani
- Athens Microneurosurgery Laboratory, Department of Neurosurgery, University of Athens, Athens, Greece
- Department of Neurosurgery, National and Kapodistrian University of Athens School of Medicine, Athens, Greece
| | - Spyridon Komaitis
- Athens Microneurosurgery Laboratory, Department of Neurosurgery, University of Athens, Athens, Greece
- Department of Neurosurgery, National and Kapodistrian University of Athens School of Medicine, Athens, Greece
| | - Danai Manolakou
- Department of Neurology, Hôpitaux Universitaires de Genève, Geneva University Hospitals, Geneva, Switzerland
| | - Despoina Chatzopoulou
- Athens Microneurosurgery Laboratory, Department of Neurosurgery, University of Athens, Athens, Greece
| | - Nikos Pantazis
- Department of Hygiene, Epidemiology and Medical Statistics, School of Medicine, National and Kapodistrian University of Athens, Athens, Greece
| | - Georgios A. Zenonos
- Department of Neurosurgery, University of Pittsburgh Medical Center, Pittsburgh, PA
| | - Constantinos G. Hadjipanayis
- Department of Neurosurgery, Icahn School of Medicine, Mount Sinai Health System, New York, NY, USA
- Department of Neurosurgery, University of Pittsburgh Medical Center, Pittsburgh, PA
- Department of Neurosurgery, Icahn School of Medicine Mount Sinai Beth Israel, Mount Sinai Health System, New York, NY, USA
| | - George Stranjalis
- Athens Microneurosurgery Laboratory, Department of Neurosurgery, University of Athens, Athens, Greece
- Department of Neurosurgery, National and Kapodistrian University of Athens School of Medicine, Athens, Greece
| | - Christos Koutsarnakis
- Athens Microneurosurgery Laboratory, Department of Neurosurgery, University of Athens, Athens, Greece
- Department of Neurosurgery, National and Kapodistrian University of Athens School of Medicine, Athens, Greece
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Ogawa S, Sato D, Torazawa S, Ota T. Acute Thrombectomy for Contralateral Internal Carotid Artery Occlusion after Revascularization Surgery for Quasi-moyamoya Disease: A Case Report. NMC Case Rep J 2021; 8:673-679. [PMID: 35079533 PMCID: PMC8769482 DOI: 10.2176/nmccrj.cr.2021-0089] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/18/2021] [Accepted: 06/29/2021] [Indexed: 11/27/2022] Open
Abstract
Ischemic complications can occur after revascularization surgery for moyamoya disease, but acute contralateral internal carotid artery (ICA) occlusion is an extremely rare complication. The patient was a 51-year-old woman with no medical history. Left frontal lobe infarction and bilateral ICA terminal stenosis were identified by repeated transient right paresis and aphasia. We diagnosed her with quasi-moyamoya disease associated with hyperthyroidism and performed revascularization surgery for the symptomatic left side. Although neurological symptoms did not worsen immediately after the surgery, disturbance of consciousness, right conjugate deviation, and left paresis appeared 4 hr after the surgery. New infarction appeared in the right frontal lobe, and the blood signal beyond the right middle cerebral artery (MCA) disappeared on MRI and MRA. Mechanical thrombectomy (MT) using a suction catheter improved antegrade blood flow in the MCA. The left paresis remained at discharge (modified Rankin Scale score = 4), but she was able to walk independently 3 months after the operation and was independent at home. Acute contralateral ICA occlusion after revascularization for moyamoya disease is an extremely rare complication, but the symptoms can be severe and treatment should be considered. To the best of our knowledge, there have been no reports of MT for postoperative acute contralateral ICA occlusion. Since the results of endovascular treatment such as percutaneous transluminal angioplasty and stent placement for patients with moyamoya disease are poor, MT using an aspiration catheter could be a good treatment option.
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Affiliation(s)
- Shotaro Ogawa
- Department of Neurosurgery, Tokyo Metropolitan Tama Medical Center, Fuchu, Tokyo, Japan
| | - Daisuke Sato
- Department of Neurosurgery, Tokyo Metropolitan Tama Medical Center, Fuchu, Tokyo, Japan
| | - Seiei Torazawa
- Department of Neurosurgery, Tokyo Metropolitan Tama Medical Center, Fuchu, Tokyo, Japan
| | - Takahiro Ota
- Department of Neurosurgery, Tokyo Metropolitan Tama Medical Center, Fuchu, Tokyo, Japan
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Nakamura H, Sato K, Yoshimura S, Hayashi Y, Izumo T, Tokunaga Y. Moyamoya Disease Associated with Graves' Disease and Down Syndrome: A Case Report and Literature Review. J Stroke Cerebrovasc Dis 2020; 30:105414. [PMID: 33130479 DOI: 10.1016/j.jstrokecerebrovasdis.2020.105414] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/19/2020] [Revised: 09/26/2020] [Accepted: 10/14/2020] [Indexed: 12/19/2022] Open
Abstract
BACKGROUND Moyamoya vessels are cerebral vasculopathies characterized by net-like collateral vessel formation at the cerebral basal area and stenosis of the terminal internal carotid artery, proximal middle cerebral artery, and anterior cerebral artery. A diagnosis of Moyamoya disease depends on the bilateral presence of Moyamoya vessels. Moyamoya disease associated with Graves' disease has rarely been reported to be a cause of ischemic events due to hyperthyroidism. However, there are extremely rare cases of Moyamoya disease with concurrent Graves' disease and Down syndrome. We aimed to report such a case, and to compare these cases' clinical features, pathogenesis, and treatment effects to those of the cases of concurrent Moyamoya disease and Graves' disease alone. METHODS We performed an English literature search using the PubMed database and the keywords Moyamoya, quasi-Moyamoya, Graves' disease, thyrotoxicosis, Down syndrome, and trisomy 21. RESULTS Only five cases of Moyamoya disease with Graves' disease and Down syndrome have been reported, including our own. Four patients were female (80%), and all underwent antithyroid therapy and experienced ischemic episodes, including transient ischemic attacks. At the time of their vascular accident, two patients were in a thyrotoxic state; only our patient was in a euthyroid state. The mean age was 15.6 years (standard deviation: 6.1), which was younger than the mean age of 31.4 years (standard deviation: 13) in patients with Moyamoya disease and Graves' disease alone. Down syndrome is commonly associated with abnormal vascular networks due to increased endostatin concentrations or immunological abnormalities such as those that occur in Graves' disease. Graves' disease accelerates the progression of Moyamoya disease and ischemic attacks due to atherosclerosis, enhances sympathetic nervous system activity and immunological changes. As compared to Moyamoya disease patients, patients with concurrent Graves' disease only and Moyamoya disease patients with concurrent Graves' disease and Down syndrome may experience accelerated disease progression or more frequent ischemic attacks. CONCLUSION Early imaging follow-ups and strict control of thyroid function are necessary in such cases; if ischemic attacks have already occurred, revascularization surgery may be effective.
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Affiliation(s)
- Hikaru Nakamura
- Department of Neurosurgery, Nagasaki Prefecture Shimabara Hospital, 7895 Shimokawasiri, Shimabara, Nagasaki 855-0816, Japan.
| | - Kei Sato
- Department of Neurosurgery, Nagasaki Prefecture Shimabara Hospital, 7895 Shimokawasiri, Shimabara, Nagasaki 855-0816, Japan
| | - Shota Yoshimura
- Department of Neurosurgery, Nagasaki Prefecture Shimabara Hospital, 7895 Shimokawasiri, Shimabara, Nagasaki 855-0816, Japan
| | - Yukishige Hayashi
- Department of Neurosurgery, Nagasaki Prefecture Shimabara Hospital, 7895 Shimokawasiri, Shimabara, Nagasaki 855-0816, Japan
| | - Tsuyoshi Izumo
- Department of Neurosurgery, Nagasaki University School of Medicine, 1-7-1 Sakamoto, Nagasaki, Nagasaki 852-8501, Japan
| | - Yoshiharu Tokunaga
- Department of Neurosurgery, Nagasaki Prefecture Shimabara Hospital, 7895 Shimokawasiri, Shimabara, Nagasaki 855-0816, Japan
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Tashiro R, Niizuma K, Khor SS, Tokunaga K, Fujimura M, Sakata H, Endo H, Inoko H, Ogasawara K, Tominaga T. Identification of HLA-DRB1*04:10 allele as risk allele for Japanese moyamoya disease and its association with autoimmune thyroid disease: A case-control study. PLoS One 2019; 14:e0220858. [PMID: 31412073 PMCID: PMC6693760 DOI: 10.1371/journal.pone.0220858] [Citation(s) in RCA: 14] [Impact Index Per Article: 2.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/17/2019] [Accepted: 07/24/2019] [Indexed: 11/28/2022] Open
Abstract
Background and purpose Moyamoya disease (MMD) is a progressive cerebrovascular disease with unknown etiology. Growing evidence suggest its involvement of autoimmune and genetic mechanisms in the pathogenesis of MMD. This study aims to clarify the association between HLA allele and MMD. Methods Case-control study: the DNA of 136 MMD patients in Japan was extracted and the genotype of human leukocyte antigen (HLA) from this DNA was determined by super-high-resolution single-molecule sequence-based typing using next-generation sequencing. Next, the frequency of each HLA allele (HLA-A, HLA-B, HLA-C, HLA-DRB1, HLA-DQB1, and HLA-DPB1) was compared with those in the Japanese control database. In addition, haplotype estimation was performed using the expectation maximization algorithm. Results The frequencies of the HLA-DRB1*04:10 allele (4.77% vs. 1.47% in the control group; P = 1.7 × 10−3; odds ratio [OR] = 3.35) and of the HLA-DRB1*04:10–HLA-DQB1*04:02 haplotype (haplotype frequency 4.41% vs. 1.35% in the control group; P = 2.0 × 10−3; OR = 3.37) significantly increased. The frequency of thyroid diseases, such as Graves’ disease and Hashimoto thyroiditis, increased in HLA-DRB1*04:10-positive MMD patients compared with that in HLA-DRB1*04:10-negative MMD patients. Conclusions HLA-DRB1*04:10 is a risk allele and HLA-DRB1*04:10–HLA-DQB1*04:02 a risk haplotype for MMD. In addition, HLA-DRB1*04:10 is associated with thyroid disease in MMD patients.
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Affiliation(s)
- Ryosuke Tashiro
- Department of Neurosurgery, Tohoku University Graduate School of Medicine, Sendai, Japan
- Department of Immunobiology, Tohoku University Institute of Development, Aging and Cancer, Sendai, Japan
| | - Kuniyasu Niizuma
- Department of Neurosurgery, Tohoku University Graduate School of Medicine, Sendai, Japan
- Department of Neurosurgical Engineering and Translational Neuroscience, Graduate School of Biomedical Engineering, Tohoku University, Sendai, Japan
- Department of Neurosurgical Engineering and Translational Neuroscience, Tohoku University Graduate School of Medicine, Sendai, Japan
- * E-mail:
| | - Seik-Soon Khor
- Department of Human Genetics, the University of Tokyo Graduate School of Medicine, Tokyo, Japan
| | - Katsushi Tokunaga
- Department of Human Genetics, the University of Tokyo Graduate School of Medicine, Tokyo, Japan
| | - Miki Fujimura
- Department of Neurosurgery, Kohnan Hospital, Sendai, Japan
| | - Hiroyuki Sakata
- Department of Neurosurgery, Tohoku University Graduate School of Medicine, Sendai, Japan
| | - Hidenori Endo
- Department of Neurosurgery, Tohoku University Graduate School of Medicine, Sendai, Japan
| | | | - Koetsu Ogasawara
- Department of Immunobiology, Tohoku University Institute of Development, Aging and Cancer, Sendai, Japan
| | - Teiji Tominaga
- Department of Neurosurgery, Tohoku University Graduate School of Medicine, Sendai, Japan
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Choi J, Suthakar P, Farmand F. Fatal outcome in a Hispanic woman with moyamoya syndrome and Graves' disease. Endocrinol Diabetes Metab Case Rep 2016; 2016:EDM160045. [PMID: 27857839 PMCID: PMC5097141 DOI: 10.1530/edm-16-0045] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/04/2016] [Accepted: 10/24/2016] [Indexed: 11/08/2022] Open
Abstract
We describe the case of a young Hispanic female who presented with thyrotoxicosis with seizures and ischemic stroke. She was diagnosed with a rare vasculopathy - moyamoya syndrome. After starting antithyroid therapy, her neurologic symptoms did not improve. Acute neurosurgical intervention had relieved her symptoms in the immediate post-operative period after re-anastomosis surgery. However, 2 post-operative days later, she was found to be in status epilepticus and in hyperthyroid state. She quickly deteriorated clinically and had expired a few days afterward. This is the second case in literature of a fatality in a patient with moyamoya syndrome and Graves' disease. However, unlike the other case report, our patient had undergone successful revascularization surgery. We believe her underlying non-euthyroid state had potentiated her clinical deterioration. Case studies have shown positive correlation between uncontrolled hyperthyroidism and stroke-like symptoms in moyamoya syndrome. Mostly all patients with these two disease processes become symptomatic in marked hyperthyroid states. Thus, it may be either fluctuations in baseline thyroid function or thyrotoxicosis that potentiate otherwise asymptomatic moyamoya vasculopathy. LEARNING POINTS Awareness of the association between Graves' disease and moyamoya syndrome in younger patients presenting with stroke-like symptoms.Obtaining euthyroid states before undergoing revascularization surgery may protect the patient from perioperative mortality and morbidity.Although moyamoya disease is usually thought to be genetically associated, there are reports that thyroid antibodies may play a role in its pathogenesis and have an autoimmune link.Fluctuations in baseline thyroid function for patients with known Graves' disease may be a potentiating factor in exacerbating moyamoya vasculopathy.
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Affiliation(s)
| | - Perin Suthakar
- Neurology , Arrowhead Regional Medical Center, Colton, California , USA
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Acute Thyrotoxicosis of Graves Disease Associated with Moyamoya Vasculopathy and Stroke in Latin American Women: A Case Series and Review of the Literature. World Neurosurg 2016; 92:95-107. [PMID: 27163552 DOI: 10.1016/j.wneu.2016.04.122] [Citation(s) in RCA: 9] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/12/2016] [Accepted: 04/29/2016] [Indexed: 11/22/2022]
Abstract
OBJECTIVE Moyamoya disease is a cerebral vasculopathy characterized by stenosis of the terminal internal carotid artery, proximal middle cerebral artery, and anterior cerebral artery. There is an association between moyamoya vasculopathy and Graves disease, primarily in Asian populations. Here, we present the largest series of non-Asian, predominantly Latino patients with moyamoya vasculopathy in the setting of Graves thyrotoxicosis, as well as the largest review of the literature to date. METHODS We retrospectively analyzed patients presenting with stroke in the setting of clinical Graves disease to our institution from 2004 to 2014. Moyamoya vasculopathy was diagnosed by magnetic resonance angiography in all patients. RESULTS Eight patients with Graves disease thyrotoxicosis and moyamoya vasculopathy were identified. Six patients were effectively managed with aggressive medical management using antithyroid and antiplatelet medications. No recurrent strokes were noted once thyrotoxicosis was controlled. Intracranial bypass was necessary in 2 patients who failed medical management. Seventy-nine additional cases were reported from the literature. There was no significant difference in clinical improvement between medical therapy alone and medical therapy with neurosurgical prophylaxis (87.0% vs. 88.0%, respectively; P = 0.94). CONCLUSIONS Moyamoya vasculopathy associated with Graves disease thyrotoxicosis in non-Asian women may be more common than previously thought. In addition, our series suggests that thyrotoxicosis promotes the progression of vasculopathy. Based on our review, there is no significant difference in clinical improvement between proper medical and surgical therapies. Aggressive medical therapy should be considered first-line treatment for moyamoya vasculopathy with Graves thyrotoxicosis, with neurosurgical rescue reserved for medically refractory cases.
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Shimogawa T, Morioka T, Sayama T, Hamamura T, Yasuda C, Arakawa S. Champagne bottle neck sign in a patient with Moyamoya syndrome. World J Clin Cases 2014; 2:474-477. [PMID: 25232554 PMCID: PMC4163773 DOI: 10.12998/wjcc.v2.i9.474] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/12/2014] [Revised: 03/20/2014] [Accepted: 07/15/2014] [Indexed: 02/05/2023] Open
Abstract
The champagne bottle neck (CBN) sign refers to a reduction in the diameter of the proximal portion of the internal carotid artery that resembles a CBN, and is a characteristic feature of Moyamoya disease. A 43-year-old woman with an infarction of the posterior limb of the left internal capsule was diagnosed with Moyamoya syndrome associated with Graves’ disease. The CBN sign was observed bilaterally. Cerebral revascularization surgery was performed, including left-sided superficial temporal artery to middle cerebral artery anastomosis. During four years of follow-up, she maintained a euthyroid state and did not have any further cerebral ischemic events. The CBN signs remained unchanged on both sides during this time. This is the first report of the CBN sign in a patient with Moyamoya syndrome associated with Graves’ disease.
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Ishigami A, Toyoda K, Suzuki R, Miyashita F, Iihara K, Minematsu K. Neurologic improvement without angiographic improvement after antithyroid therapy in a patient with Moyamoya syndrome. J Stroke Cerebrovasc Dis 2013; 23:1256-8. [PMID: 24119625 DOI: 10.1016/j.jstrokecerebrovasdis.2013.08.025] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/25/2013] [Revised: 08/26/2013] [Accepted: 08/28/2013] [Indexed: 11/19/2022] Open
Abstract
Moyamoya disease with special complications, including Graves' disease, is called as moyamoya syndrome. A 22-year-old Japanese woman had left middle cerebral artery (MCA) territory infarction complicated with Graves' disease. She had right-sided hemiparesis that deteriorated on day 8 with the infarct growth and thyrotoxicosis. On angiogram, the left MCA was occluded at the origin without moyamoya vessels. Positron emission tomography (PET) revealed misery-perfusion phenomenon in the left MCA territory. After initiation of the antithyroid therapy, her hemiparesis became milder. Seventeen months later, her thyroid function was normalized and net-like collateral moyamoya vessels proliferated in the left MCA territory. Misery-perfusion phenomenon persisted on PET. This report is unique in the point of neurologic recovery of the moyamoya patient right after initiation of antithyroid medication without radiological improvement.
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Affiliation(s)
- Akiko Ishigami
- Department of Cerebrovascular Medicine, National Cerebral and Cardiovascular Center, Suita, Osaka, Japan.
| | - Kazunori Toyoda
- Department of Cerebrovascular Medicine, National Cerebral and Cardiovascular Center, Suita, Osaka, Japan
| | - Rieko Suzuki
- Department of Cerebrovascular Medicine, National Cerebral and Cardiovascular Center, Suita, Osaka, Japan
| | - Fumio Miyashita
- Department of Cerebrovascular Medicine, National Cerebral and Cardiovascular Center, Suita, Osaka, Japan
| | - Koji Iihara
- Department of Neurosurgery, National Cerebral and Cardiovascular Center, Suita, Osaka, Japan
| | - Kazuo Minematsu
- Department of Cerebrovascular Medicine, National Cerebral and Cardiovascular Center, Suita, Osaka, Japan
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Li D, Yang W, Xian P, Liu P, Bao X, Zong R, Duan L. Coexistence of moyamoya and Graves' diseases: the clinical characteristics and treatment effects of 21 Chinese patients. Clin Neurol Neurosurg 2013; 115:1647-52. [PMID: 23518423 DOI: 10.1016/j.clineuro.2013.02.018] [Citation(s) in RCA: 12] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/15/2012] [Revised: 02/15/2013] [Accepted: 02/18/2013] [Indexed: 02/07/2023]
Abstract
OBJECTIVE To investigate the clinical features, pathogenesis and treatment effect of patients who had moyamoya disease (MMD) concurrent with Graves' disease (GD). PATIENTS AND METHODS Retrospective analysis of the examination and treatment results of the MMD patients with GD from January 2003 to April 2012 treated by our teamwork. RESULTS There were 1493 MMD patients included in this retrospective study. Among them, there were 16 female and 5 male MMD patients coexisted with Graves' diseases, and 15 of them were adult patients. Bilateral and unilateral lesions were observed in 18 and 3 cases, respectively. 19 patients show cerebral infarction. 18 patients manifested MMD symptoms when they were in a hyperthyroid state. 17 patients received encephaloduroarteriosynangiosis (EDAS) surgery after antithyroid treatment. There were 20 patients completed 2 and 77 months follow-up. 17 patients had good recovery and the other 3 patients had mild or moderate disability. Post-operative DSA achieved in 8 patients examinations showed good collateral circulation to the brain from superficial temporal artery (STA). CONCLUSION Moyamoya disease complicating GD is mostly occurred in adult female patients. Their clinical symptoms are mainly ischemic lesions and usually occur during the state of hyperthyroidism. The pathogenesis of moyamoya disease complicating GD may be associated with a variety of genetic and immune factors. Surgical treatment can establish effective collateral circulation and reduce the risk of recurrence of stroke.
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Affiliation(s)
- Desheng Li
- Department of Neurosurgery, Affiliated Hospital of Academy of Military Medical Sciences, No 8 Dongdajie, Beijing 100071, China
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